A 3-year-old boy was admitted for yellowish discharge from a tiny opening in the midline prepubic area on the dorsal base of the penis. No other symptom related to the genitourinary tract was noted.Sinuousgraphy showed a non-communicating sinus tract with the urinary tract. At surgical exploration, through a diamond-shaped incision, a 4.5 cm-long sinus was found that ended blindly as a fibrous cord at the anterior surface of the pubic symphisis. Histologically, it was lined by stratified squamous epithelium and surrounded by bundles of smooth muscle and collagen. According to Stephens' classification, the sinus appears to be a variant of dorsal urethral duplication of Stephens type 3.